- Email: [email protected]
Cerebral embolism and carotid-hyoid impingement syndrome
Case Reports / Journal of Clinical Neuroscience 64 (2019) 27–29 [12] Fischer S, Weber J, Senn-Schonenberger I, Cerny T, Hundsberger T. Neuroborreliosis mimicking leptomeningeal carcinomatosis in a patient with breast cancer: a case report. J Investig Med High Impact Case Rep 2014;2. 2324709614529417. [13] Nelson JA, Wolf MD, Yuh WT, Peeples ME. Cranial nerve involvement with Lyme borreliosis demonstrated by magnetic resonance imaging. Neurology 1992;42:671–3.
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[14] Pachner AR, Delaney E, O’Neill T, Major E. Inoculation of nonhuman primates with the N40 strain of Borrelia burgdorferi leads to a model of Lyme neuroborreliosis faithful to the human disease. Neurology 1995;45:165–72. [15] Topakian R, Stieglbauer K, Nussbaumer K, Aichner FT. Cerebral vasculitis and stroke in Lyme neuroborreliosis. Two case reports and review of current knowledge. Cerebrovasc Dis 2008;26:455–61.
https://doi.org/10.1016/j.jocn.2019.03.048
Cerebral embolism and carotid-hyoid impingement syndrome Lay Kun Kho a,b,⇑, Timothy R Bates b, Andrew Thompson c, Ferry Dharsono c, David Prentice a a b c
Royal Perth Hospital, Western Australia, Australia Midland St John Of God Hospital, Western Australia, Australia Neurological Intervention & Imaging Service of WA (NIISwa), Western Australia, Australia
a r t i c l e
i n f o
Article history: Received 1 August 2018 Accepted 22 March 2019
Keywords: Embolic stroke Hyoid bone Carotid-hyoid syndrome
a b s t r a c t Embolic stroke of undetermined source (ESUS) is not uncommon in young patients. Here, we describe two cases of stroke from an unusual aetiology; cerebral embolization from carotid artery injury presumed secondary to hyoid bone impingement. Both patients demonstrated angiographic evidence of hyoid bone impingement. Following resection of the greater cornu of the hyoid bone, neither patient had further strokes. Ó 2019 Published by Elsevier Ltd.
1. Introduction Embolic stroke of undetermined source (ESUS) is not uncommon in young patients. Extensive investigations may not determine the cause of stroke [1]. This uncertainty is distressing for patients and frustrating for clinicians. We describe 2 cases with an unusual cause of stroke; that being cerebral emboli from carotid artery injury secondary to presumed impingement from the hyoid bone. 2. Case reports 2.1. Case 1 29-year old woman presented with intermittent episodes of expressive dysphasia for six weeks. Two weeks prior, she had uncomplicated drainage of left upper incisor dental abscess. On examination, she was afebrile, normotensive with normal neurological examination. MRI showed multiple infarcts in left middle and anterior cerebral artery territories with her left internal carotid artery (ICA) in normal position i.e. anterolateral to external carotid artery (ECA). Subsequent CT angiogram (CTA) showed minor irregularities of the proximal left ICA but the ICA was now postero-medial to ECA and behind the greater cornu of the hyoid (Fig. 1). Extensive workup (Transthoracic Echocardiogram, Transoesophageal Echocardiogram, Holter, thrombophilia, lumbar puncture and autoimmune screens) was normal. She was discharged on Aspirin 100 mg/day. Five months later she presented with right arm weakness and dysarthria lasting 30 min. She also noticed four episodes of right ⇑ Corresponding author at: Department of Neurology, Level 7, Royal Perth Hospital, Wellington St, Perth, WA 6000, Australia. E-mail addresses: [email protected] (L.K. Kho), [email protected] (T.R Bates), [email protected] (A. Thompson), Ferry.Dharsono@ health.wa.gov.au (F. Dharsono), [email protected] (D. Prentice).
leg weakness lasting several minutes over the preceding two months. This time, her MRI showed new areas of infarction on DWI sequences in left MCA territory. Further history revealed that prior to the first stroke, the patient had developed a nervous habit of ‘‘clicking her neck” by rapid rotation from left to right, up to ten times a day for three months. The second presentation to hospital occurred after falling asleep on the couch for eight hours lying on her left side in an awkward position. She proceeded to a cerebral angiogram with head repositioning. On extreme left lateral head tilt, there was medial subluxation of the left ICA to behind the greater cornu of the hyoid bone. She was commenced on Clopidogrel 75 mg/day. An otolaryngologist was consulted and resection of the left greater cornu of the hyoid was performed. At operation a dense fibrous band connected the tip of the hyoid to superior cornu of thyroid cartilage. Histopathology was normal. She has had no recurrent events.
2.2. Case 2 A 34-year old woman presented with sudden onset dysarthria and left facial droop. In the preceding fortnight, she reported a constant right earache that previously had been intermittent for two years. This pain radiated to the mid-sternomastoid area. It was unchanged by deglutition or phonation, but was worse in cold weather or pressure over the hyoid area. There was no history of neck injury or manipulation, with no known conventional vascular risk factors. Her only medication was the oral contraceptive pill. Initial MRI revealed DWI hyperintensities consistent with multiple infarcts in the right frontal lobe. Contrast-enhanced CTA showed a shelf-like protuberance from the medial wall of the right carotid artery, just above the carotid bifurcation (Fig. 2). Extensive workup for alternative causes of stroke was unremarkable. Patient was referred for vessel-wall imaging with head repositioning due to the neck pain and close proximity of the hyoid bone. This showed the right ICA to be hypermobile. In the neutral
28
Case Reports / Journal of Clinical Neuroscience 64 (2019) 27–29
Fig. 1. (A) MRI confirmed acute discrete left frontal and temporal infarcts on axial DWI. (B) CTA: Initial axial image demonstrated medial displacement and rotation of the ICA (arrow). (C) Axial 2D time-of-flight MRA demonstrating the now normal anatomic position of the ICA.
Fig. 2. (A) CTA: Initial axial image demonstrated medial displacement and rotation of the ICA with luminal filling defect (arrow). (B) MRI: Axial DWI confirmed acute right frontal opercular infarct.
position, the right ICA was subluxed medially but upon right head turning, it flipped to its usual expected position (Fig. 3). The right ICA showed eccentric medially-directed intimal thickening. Aspirin prophylaxis was commenced. In the following 12 months the ear pain persisted despite no new strokes. Due to persistent ear pain and perceived risk of further stroke, the patient underwent resection of right greater cornu of the hyoid bone with resolution of right ear pain and no further neurological issues. 3. Discussion These cases describe a rare but important cause of cryptogenic stroke. Anatomically, the carotid bifurcations are commonly at the level of C3/4 (superior border of the thyroid laminae) [2], with the ICA located posterolateral to the ECA [3]. The ICA may be injured due to impingement and mobility around the greater cornu of the hyoid. Recent reports of stroke have documented carotid artery injury from excessive head rotation or lateral flexion [4–6]. The carotid may even become entrapped behind the hyoid. The proximity of both the greater cornu of the hyoid and the styloid process have been shown to be a risk factor for stroke. It is likely that mechanical
trauma can lead to vessel-wall inflammation, dissection or injury with subsequent thrombosis and embolism. Recently, it has been shown that swallowing also can induce medial displacement of the carotid artery and arterial injury due to hyoid bone impingement [7]. Clues that may alert the clinician to this condition include repetitive neck movements, prolonged neck postures or new onset neck pain over the ICA origin. Our second patient described ear pain suggestive of glossopharyngeal neuralgia without throat pain and syncope. As the hyoid arises embryogenically from the third pharyngeal arch it is conceivable that the pain originated from the periosteum of the hyoid. The hyoid bone pain syndrome has been recognised by otolaryngologists for decades [8]. Diagnosis of this condition requires a high index of suspicion. Carotid ultrasound has been used successfully to image the relationship of the carotid artery to the hyoid bone on head rotation and swallowing [7] although MRI and CTA are less operatordependent. CTA may provide an initial screen for this condition. Cervical flexion with repeat ultrasound or CTA images may provide the best diagnostic imaging to confirm clinically suspected cases. The management is uncertain. Most reported cases have undergone resection of the greater cornu. This approach seems reason-
Case Reports / Journal of Clinical Neuroscience 64 (2019) 29–32
29
Fig. 3. (A) Subsequent Axial 2D time-of-flight MRA, demonstrated persistent medial displacement and rotation of the ICA (arrow) with the head in a neutral position. (B) MRI: Axial T1-weighted imaging, head turned to the right, demonstrated restoration of the normal anatomic position of the ICA (arrow).
able considering the minimal morbidity and allows the patient physical and psychological freedom with neck movement. 4. Conclusion Hyoid bone impingement of the carotid artery should be considered in stroke of indeterminate aetiology especially in the presence of neck, throat, ear pain, repetitive neck movements and prolonged neck position. CTA neck with head rotation may be used for initial screening. Appendix A. Supplementary data Supplementary data to this article can be found online at https://doi.org/10.1016/j.jocn.2019.03.040.
References [1] Hart RG, Catanese L, Perera KS, et al. Embolic Stroke of Undertermined Source: A systematic review and clinical update. Stroke 2017;48(4):867–72. [2] Mirjalili SA, McFadden SL, Buckenham T, et al. Vertebral levels of key landmarks in the neck. Clin Anat 2012;25(7):851–7. [3] Lee W. General principles of carotid Doppler ultrasonography. Ultrasonography 2014;33(1):11–7. [4] Ludt C, Leppert M, Jones A, et al. Multiple strokes associated with elongation of the hyoid bone. Neurohospitalist 2018;8(1):35–7. [5] Renard D, Rougier M, Aichoun I, et al. Hyoid bone-related focal carotid vasculopathy. J Neurol 2011;258:1540–1. [6] Renard D, Freitag C. Hyoid-related internal carotid artery dissection. J Neurol 2012;259:2501–2. [7] Kinoshita N, Saito K, Yamaguchi Y, et al. Flip-flop phenomenon: swallowinginduced arterial displacement as an indicator of carotid artery disease. Cerebrovasc Dis 2018;45(5–6):258–62. [8] Robinson PJ, Davis JP, Fraser JG. The hyoid syndrome: a pain in the neck. J Laryngol Otol 1994;108:855–8.
https://doi.org/10.1016/j.jocn.2019.03.040
Mirror writing after perimesencephalic subarachnoid haemorrhage Benjamin Fleming a,⇑, Alexios A. Adamides a,b a b
Department of Neurosurgery, The Royal Melbourne Hospital, Parkville, Victoria, Australia Department of Surgery, University of Melbourne, Victoria, Australia
a r t i c l e
i n f o
Article history: Received 13 March 2019 Accepted 21 March 2019
Keywords: Mirror-writing Mirror writing Perimesencephalic subarachnoid hemorrhage Non-aneurysmal subarachnoid hemorrhage Delayed ischemic neurological deficit Hyponatremia
a b s t r a c t We report a case of a right-handed Caucasian woman who developed mirror writing following a nonaneurysmal, non-traumatic subarachnoid haemorrhage. The patient was unaware of having this phenomenon, and it was detected by clinical staff when the patient was writing a card to a family member. Serial imaging has ruled out a stroke as well as an underlying vascular abnormality. This phenomenon resolved after two months. Whilst there is a body of literature surrounding mirror writing, to our knowledge, this is the first description of mirror writing in the setting of a perimesencephalic subarachnoid haemorrhage. Crown Copyright Ó 2019 Published by Elsevier Ltd. All rights reserved.
⇑ Corresponding author at: Department of Neurosurgery, The Royal Melbourne Hospital, 300 Grattan Street, Parkville, Victoria 3050, Australia. E-mail address: [email protected] (B. Fleming).
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[14] Pachner AR, Delaney E, O’Neill T, Major E. Inoculation of nonhuman primates with the N40 strain of Borrelia burgdorferi leads to a model of Lyme neuroborreliosis faithful to the human disease. Neurology 1995;45:165–72. [15] Topakian R, Stieglbauer K, Nussbaumer K, Aichner FT. Cerebral vasculitis and stroke in Lyme neuroborreliosis. Two case reports and review of current knowledge. Cerebrovasc Dis 2008;26:455–61.
https://doi.org/10.1016/j.jocn.2019.03.048
Cerebral embolism and carotid-hyoid impingement syndrome Lay Kun Kho a,b,⇑, Timothy R Bates b, Andrew Thompson c, Ferry Dharsono c, David Prentice a a b c
Royal Perth Hospital, Western Australia, Australia Midland St John Of God Hospital, Western Australia, Australia Neurological Intervention & Imaging Service of WA (NIISwa), Western Australia, Australia
a r t i c l e
i n f o
Article history: Received 1 August 2018 Accepted 22 March 2019
Keywords: Embolic stroke Hyoid bone Carotid-hyoid syndrome
a b s t r a c t Embolic stroke of undetermined source (ESUS) is not uncommon in young patients. Here, we describe two cases of stroke from an unusual aetiology; cerebral embolization from carotid artery injury presumed secondary to hyoid bone impingement. Both patients demonstrated angiographic evidence of hyoid bone impingement. Following resection of the greater cornu of the hyoid bone, neither patient had further strokes. Ó 2019 Published by Elsevier Ltd.
1. Introduction Embolic stroke of undetermined source (ESUS) is not uncommon in young patients. Extensive investigations may not determine the cause of stroke [1]. This uncertainty is distressing for patients and frustrating for clinicians. We describe 2 cases with an unusual cause of stroke; that being cerebral emboli from carotid artery injury secondary to presumed impingement from the hyoid bone. 2. Case reports 2.1. Case 1 29-year old woman presented with intermittent episodes of expressive dysphasia for six weeks. Two weeks prior, she had uncomplicated drainage of left upper incisor dental abscess. On examination, she was afebrile, normotensive with normal neurological examination. MRI showed multiple infarcts in left middle and anterior cerebral artery territories with her left internal carotid artery (ICA) in normal position i.e. anterolateral to external carotid artery (ECA). Subsequent CT angiogram (CTA) showed minor irregularities of the proximal left ICA but the ICA was now postero-medial to ECA and behind the greater cornu of the hyoid (Fig. 1). Extensive workup (Transthoracic Echocardiogram, Transoesophageal Echocardiogram, Holter, thrombophilia, lumbar puncture and autoimmune screens) was normal. She was discharged on Aspirin 100 mg/day. Five months later she presented with right arm weakness and dysarthria lasting 30 min. She also noticed four episodes of right ⇑ Corresponding author at: Department of Neurology, Level 7, Royal Perth Hospital, Wellington St, Perth, WA 6000, Australia. E-mail addresses: [email protected] (L.K. Kho), [email protected] (T.R Bates), [email protected] (A. Thompson), Ferry.Dharsono@ health.wa.gov.au (F. Dharsono), [email protected] (D. Prentice).
leg weakness lasting several minutes over the preceding two months. This time, her MRI showed new areas of infarction on DWI sequences in left MCA territory. Further history revealed that prior to the first stroke, the patient had developed a nervous habit of ‘‘clicking her neck” by rapid rotation from left to right, up to ten times a day for three months. The second presentation to hospital occurred after falling asleep on the couch for eight hours lying on her left side in an awkward position. She proceeded to a cerebral angiogram with head repositioning. On extreme left lateral head tilt, there was medial subluxation of the left ICA to behind the greater cornu of the hyoid bone. She was commenced on Clopidogrel 75 mg/day. An otolaryngologist was consulted and resection of the left greater cornu of the hyoid was performed. At operation a dense fibrous band connected the tip of the hyoid to superior cornu of thyroid cartilage. Histopathology was normal. She has had no recurrent events.
2.2. Case 2 A 34-year old woman presented with sudden onset dysarthria and left facial droop. In the preceding fortnight, she reported a constant right earache that previously had been intermittent for two years. This pain radiated to the mid-sternomastoid area. It was unchanged by deglutition or phonation, but was worse in cold weather or pressure over the hyoid area. There was no history of neck injury or manipulation, with no known conventional vascular risk factors. Her only medication was the oral contraceptive pill. Initial MRI revealed DWI hyperintensities consistent with multiple infarcts in the right frontal lobe. Contrast-enhanced CTA showed a shelf-like protuberance from the medial wall of the right carotid artery, just above the carotid bifurcation (Fig. 2). Extensive workup for alternative causes of stroke was unremarkable. Patient was referred for vessel-wall imaging with head repositioning due to the neck pain and close proximity of the hyoid bone. This showed the right ICA to be hypermobile. In the neutral
28
Case Reports / Journal of Clinical Neuroscience 64 (2019) 27–29
Fig. 1. (A) MRI confirmed acute discrete left frontal and temporal infarcts on axial DWI. (B) CTA: Initial axial image demonstrated medial displacement and rotation of the ICA (arrow). (C) Axial 2D time-of-flight MRA demonstrating the now normal anatomic position of the ICA.
Fig. 2. (A) CTA: Initial axial image demonstrated medial displacement and rotation of the ICA with luminal filling defect (arrow). (B) MRI: Axial DWI confirmed acute right frontal opercular infarct.
position, the right ICA was subluxed medially but upon right head turning, it flipped to its usual expected position (Fig. 3). The right ICA showed eccentric medially-directed intimal thickening. Aspirin prophylaxis was commenced. In the following 12 months the ear pain persisted despite no new strokes. Due to persistent ear pain and perceived risk of further stroke, the patient underwent resection of right greater cornu of the hyoid bone with resolution of right ear pain and no further neurological issues. 3. Discussion These cases describe a rare but important cause of cryptogenic stroke. Anatomically, the carotid bifurcations are commonly at the level of C3/4 (superior border of the thyroid laminae) [2], with the ICA located posterolateral to the ECA [3]. The ICA may be injured due to impingement and mobility around the greater cornu of the hyoid. Recent reports of stroke have documented carotid artery injury from excessive head rotation or lateral flexion [4–6]. The carotid may even become entrapped behind the hyoid. The proximity of both the greater cornu of the hyoid and the styloid process have been shown to be a risk factor for stroke. It is likely that mechanical
trauma can lead to vessel-wall inflammation, dissection or injury with subsequent thrombosis and embolism. Recently, it has been shown that swallowing also can induce medial displacement of the carotid artery and arterial injury due to hyoid bone impingement [7]. Clues that may alert the clinician to this condition include repetitive neck movements, prolonged neck postures or new onset neck pain over the ICA origin. Our second patient described ear pain suggestive of glossopharyngeal neuralgia without throat pain and syncope. As the hyoid arises embryogenically from the third pharyngeal arch it is conceivable that the pain originated from the periosteum of the hyoid. The hyoid bone pain syndrome has been recognised by otolaryngologists for decades [8]. Diagnosis of this condition requires a high index of suspicion. Carotid ultrasound has been used successfully to image the relationship of the carotid artery to the hyoid bone on head rotation and swallowing [7] although MRI and CTA are less operatordependent. CTA may provide an initial screen for this condition. Cervical flexion with repeat ultrasound or CTA images may provide the best diagnostic imaging to confirm clinically suspected cases. The management is uncertain. Most reported cases have undergone resection of the greater cornu. This approach seems reason-
Case Reports / Journal of Clinical Neuroscience 64 (2019) 29–32
29
Fig. 3. (A) Subsequent Axial 2D time-of-flight MRA, demonstrated persistent medial displacement and rotation of the ICA (arrow) with the head in a neutral position. (B) MRI: Axial T1-weighted imaging, head turned to the right, demonstrated restoration of the normal anatomic position of the ICA (arrow).
able considering the minimal morbidity and allows the patient physical and psychological freedom with neck movement. 4. Conclusion Hyoid bone impingement of the carotid artery should be considered in stroke of indeterminate aetiology especially in the presence of neck, throat, ear pain, repetitive neck movements and prolonged neck position. CTA neck with head rotation may be used for initial screening. Appendix A. Supplementary data Supplementary data to this article can be found online at https://doi.org/10.1016/j.jocn.2019.03.040.
References [1] Hart RG, Catanese L, Perera KS, et al. Embolic Stroke of Undertermined Source: A systematic review and clinical update. Stroke 2017;48(4):867–72. [2] Mirjalili SA, McFadden SL, Buckenham T, et al. Vertebral levels of key landmarks in the neck. Clin Anat 2012;25(7):851–7. [3] Lee W. General principles of carotid Doppler ultrasonography. Ultrasonography 2014;33(1):11–7. [4] Ludt C, Leppert M, Jones A, et al. Multiple strokes associated with elongation of the hyoid bone. Neurohospitalist 2018;8(1):35–7. [5] Renard D, Rougier M, Aichoun I, et al. Hyoid bone-related focal carotid vasculopathy. J Neurol 2011;258:1540–1. [6] Renard D, Freitag C. Hyoid-related internal carotid artery dissection. J Neurol 2012;259:2501–2. [7] Kinoshita N, Saito K, Yamaguchi Y, et al. Flip-flop phenomenon: swallowinginduced arterial displacement as an indicator of carotid artery disease. Cerebrovasc Dis 2018;45(5–6):258–62. [8] Robinson PJ, Davis JP, Fraser JG. The hyoid syndrome: a pain in the neck. J Laryngol Otol 1994;108:855–8.
https://doi.org/10.1016/j.jocn.2019.03.040
Mirror writing after perimesencephalic subarachnoid haemorrhage Benjamin Fleming a,⇑, Alexios A. Adamides a,b a b
Department of Neurosurgery, The Royal Melbourne Hospital, Parkville, Victoria, Australia Department of Surgery, University of Melbourne, Victoria, Australia
a r t i c l e
i n f o
Article history: Received 13 March 2019 Accepted 21 March 2019
Keywords: Mirror-writing Mirror writing Perimesencephalic subarachnoid hemorrhage Non-aneurysmal subarachnoid hemorrhage Delayed ischemic neurological deficit Hyponatremia
a b s t r a c t We report a case of a right-handed Caucasian woman who developed mirror writing following a nonaneurysmal, non-traumatic subarachnoid haemorrhage. The patient was unaware of having this phenomenon, and it was detected by clinical staff when the patient was writing a card to a family member. Serial imaging has ruled out a stroke as well as an underlying vascular abnormality. This phenomenon resolved after two months. Whilst there is a body of literature surrounding mirror writing, to our knowledge, this is the first description of mirror writing in the setting of a perimesencephalic subarachnoid haemorrhage. Crown Copyright Ó 2019 Published by Elsevier Ltd. All rights reserved.
⇑ Corresponding author at: Department of Neurosurgery, The Royal Melbourne Hospital, 300 Grattan Street, Parkville, Victoria 3050, Australia. E-mail address: [email protected] (B. Fleming).